Myoclonus-dystonia syndrome: epsilon-sarcoglycan mutations and phenotype

Ann Neurol. 2002 Oct;52(4):489-92. doi: 10.1002/ana.10325.

Abstract

Mutations in the gene for epsilon-sarcoglycan (SGCE) have been found to cause myoclonus-dystonia syndrome. We now report clinical and genetic findings in nine additional European families with myoclonus-dystonia syndrome. The clinical presentation in 24 affecteds was homogeneous with myoclonus predominantly of neck and upper limbs in 23 of them and dystonia, presenting as cervical dystonia and/or writer's cramp, in 13 cases. Six novel and one previously known heterozygous SGCE mutations were identified. SGCE deficiency seems to be the common pathogenetic mechanism in myoclonus-dystonia syndrome.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Child, Preschool
  • Cytoskeletal Proteins / genetics*
  • DNA Mutational Analysis
  • Dystonic Disorders / genetics*
  • Female
  • Genomic Imprinting
  • Genotype
  • Humans
  • Infant
  • Male
  • Membrane Glycoproteins / genetics*
  • Mutation
  • Myoclonus / genetics*
  • Penetrance
  • Phenotype
  • Sarcoglycans
  • Sex Factors

Substances

  • Cytoskeletal Proteins
  • Membrane Glycoproteins
  • Sarcoglycans